Geraldine Foley, Virpi Timonen and Orla Hardiman
Trinity College Dublin, Ireland
Understanding psycho-social processes underpinning engagement with
services in motor neurone disease: A qualitative study
Published in Palliative Medicine. 2014, 28(4), 318-325
Abstract
Background: People with motor neurone disease access health care services from
disease onset to end-of-life care but there has been paucity of research on how
people with motor neurone disease understand and use health care services.
Aim: To identify key psycho-social processes that underpin how people with motor
neurone disease engage with health care services.
Design: Grounded theory approach comprising in-depth qualitative interviews. Data
were collected and analysed using open, axial, and selective coding procedures.
Setting / participants: 34 people with motor neurone disease were recruited from
the Irish motor neurone disease population-based register.
Results: We identified that control, reassurance, resignation, and trust, are key
variables that shape how people with motor neurone disease engage with
healthcare services. Participants exerted control in care to cope with loss. Most
participants were resigned to death and sought reassurances from healthcare
professionals about end-of-life care. Participants questioned the benefit of lifesustaining interventions in motor neurone disease and few of them associated lifesustaining interventions with palliative care. Participants trusted healthcare
professionals who reassured them about their care and who were attuned to how
they were coming to terms with loss.
Conclusions: This study identified new and important aspects of control, trust, and
reassurance which shed light on how people with motor neurone disease engage
with healthcare professionals and approach end-of-life care. People with motor
1
neurone disease exert control in care and meaningful relationships with healthcare
professionals are important to them. Some people with motor neurone disease
prefer to die without life-sustaining interventions.
Keywords
Motor neuron disease, amyotrophic lateral sclerosis, qualitative research, terminal
care, palliative care, decision making,
2
Background
Motor neurone disease (MND), known in continental Europe and the Americas as
amyotrophic lateral sclerosis (ALS), is a rare but rapidly progressive neurological
disorder characterised by motor neuron degeneration of the central nervous system
and in some cases, frontotemporal impairment.1 Life expectancy is short and over
60% of people with MND die within 1000 days of their first symptom. 2 Invasive and
noninvasive ventilation sustain life for people with MND and the care approach in
MND is palliative (i.e. alleviation of physical, psychological and existential distress)
from the point of diagnosis.3,4
A number of researchers have investigated perceptions of health care services
among people with MND5-7, their experience of services in specific domains of care811
and during particular stages of care.12 Our previous work has identified that
people with MND have expectations of a broad range of health care services but also
report dissatisfaction with health care services.13 Fallon and Foley14 have argued that
managing the care needs of people with terminal non-malignant disease is a
challenge for healthcare professionals. A lack of knowledge among healthcare
professionals about what is important to people with MND can adversely affect how
service providers respond to service users.5,6
Evidence suggests that people with MND make decisions about various aspects of
care13 but we knew little about how MND service users engage with services. We
therefore sought to identify from a MND population-based cohort, key psycho-social
processes that underpin how people with MND engage with their services.
Research design
Grounded theory is a methodology focused on generating theory from data.15 We
drew upon a grounded theory approach15 focused on identifying psycho-social
processes in relation to a particular phenomenon. We used this approach because
we sought to explain at a substantive level15 how people with motor neurone
disease engage with health care services. Our study report conforms to Malterud’s
guidelines for reporting qualitative research.16
3
Sampling procedures
We sampled participants from the Irish MND population-based register compiled by
an Irish MND research group.17 During preliminary stages of sampling, we
purposively sampled18 participants from the register in order to capture a range of
healthcare experience. We then sampled participants from the register based on
concepts that emerged in the data (i.e. theoretical sampling).15
Participants
34 people on the register participated in the study between September 2011 and
August 2012. We recruited participants across the Republic of Ireland, resulting in a
geographically diverse sample [rural (n=19), semi-urban (n=5), and urban (n=10)].
80% of MND service users in the Republic of Ireland engage at some point with the
national MND clinic.4 We captured variation in participants’ experiences across all
care levels (e.g., primary care, tertiary care) and sampled both participants (n=26)
who had had varying degrees of contact with the national clinic and participants
(n=8) who had not had contact with the national clinic. Table 1 provides data on age,
gender, and clinical profile of participants and on disease duration for the sample. All
participants had engaged with medical and supportive care. The majority of
participants (n=32) required assistance for activities of daily living and all but five
participants were using a broad range of assistive devices. Eight participants had
engaged with gastrostomy feeding and/or noninvasive ventilation. Three
participants were in the final three months of their life at the time of their
participation (as verified subsequently from death records in the MND register).
Data collection and analysis
We sent a participation information leaflet to each person who had expressed
interest in the study upon initial contact. Over the duration of the study, we invited
47 people from the register to participate. Ten people declined and three people
who had agreed to participate were subsequently unable to due to rapid
deterioration of their condition in the weeks between first contact and scheduled
interview. We obtained full informed consent from each participant.
4
The first author (GF) conducted a qualitative interview with each participant. As per
grounded theory method15, we collected and analysed data in tandem and emerging
findings guided our sampling and the questions we asked. We aimed to
conceptualise participants’ experiences from the ‘ground up’ rather than use a preprepared interview guide based on what we might have assumed parameters of
service users’ experiences to be. The first author began interviews by asking
participants about their experiences of health care services “since MND came into
[their] lives”. She then integrated questions to expand on and verify emerging
findings.
All interviews were digitally audio-recorded and transcribed. Duration of interviews
ranged between 40 minutes and 2 hours 10 minutes; the average duration was 1
hour and 20 minutes. All participants who lived at home (n=32) chose to be
interviewed in their home. The remaining participants were interviewed in a hospice
(n=1) and in a nursing home (n=1). Anarthric participants (n=2) used high-tech
alternative and augmentative (AAC) devices to communicate their responses and five
out of six participants who had a severe dysarthria used low-tech AAC devices to add
to their verbal responses. Participants were provided with a copy of their transcript.
No participant wished to alter the content of their interview. We did not return to
participants to verify emerging theory owing to rapid progression of the disease for
the majority of participants.
The first author analysed the data using open, axial, and selective coding
procedures15 and the second author (VT) interrogated the analysis at all stages of
data collection and analysis. Data were broken down into discrete units of meaning
(concepts) and expanded to form categories (larger concepts) by coding for
similarities and differences in the data. The data were coded for psycho-social
processes (i.e. how and why participants thought or acted) in similar and different
contexts. In doing so, key categories (variables) that emerged from the data were
delineated from each other in terms of their properties (characteristics) and
dimensions (variation).15 Sampling ceased when new data no longer added new
5
meaning to categories.15 We continued to code selective data to refine relationships
between categories and subsequently, we identified key psycho-social processes in
the data. The first author compiled theoretical memos15 to guide sampling and to
build and refine relationships between categories. She and the third author (OH) are
experienced clinicians in ALS care. The theoretical memos were reflexive in nature
because the first author co-constructed the data.15 The qualitative software package
NVivo919 was used as a tool to store and retrieve the coded data and to link memos
to codes.
Ethics
Ethical approval to conduct this study was granted by the Beaumont Hospital Ethics
(Medical Research) Committee, Dublin, Ireland (REC ref. 10/59) and by the Research
Ethical Approval Committee at the School of Social Work and Social Policy, Trinity
College Dublin, Ireland (REAC ref. No. 298).
Findings
Here, we report on key categories (control, reassurance, resignation, and trust) that
emerged in the data and that shaped how participants engaged with health care
services. We also demonstrate relationships between categories that explain how
participants engaged with services.
Control and Reassurance
We found that participants’ primary expectations of health care services was first,
that they would engage with services when they felt ready to engage and second,
secure services at the end stage of MND. Feeling in control of care also reassured
participants:
I just think I’m not ready yet for the whole going to a wheelchair, be wheeled
into the back of a modified car and to be wheeled out … I’m processing it
[MND], I know it’s there and when I’m ready I’ll have no problem with it … I
think I’d have a fear about you know will this [MND] hurt me when I’m dying,
the final stage of the illness, people do die from it, that I just want to make
6
sure the services are going to be there and that any pain that's to be had,
that that’s all looked after. (Participant #7)
Participants needed to be in control over when they would engage with services and
needed time to process the life-altering impact of motor neurone disease before
they willingly accepted assistance from health care services. Participant #9 explained
why she had previously declined homecare services even though she had needed
assistance for personal care activities:
... because it takes a long time to get used to carers, like to have somebody
come into your house and get you up in the morning and give you a shower
and all that kind of stuff, it takes a lot to get used to that.
Needing to feel reassured by healthcare professionals about end-of-life care was
extremely important to participants. Participants were somewhat more concerned
about delivery of care at the end stage of MND than at any other stage of MND:
If you’re at that stage towards the end … whether you really want to be at
home, or whether you’re going to have to go to a nursing [home], the
different things maybe I would like … I would need to know exactly what all
the services provide then. (Participant #12)
We found that participants resigned themselves to the likelihood of becoming more
dependent on healthcare professionals as they advanced in the disease and actively
engaged with services as they neared end-of-life care. For most participants,
certainty about end-of-life care alleviated their anxiety. Participant #25 commented:
I’ve been telling everybody that I want to keep my independence. But I have
now got to the stage you know the steps of being independent are further
away … it’s been a pretty fast downward slide … I have been sorting out my
will and power of attorney and things like that. I'm very happy living here
7
[home] and I plan to stay on here … [with] [regional hospice] nurses because I
mean they do home care.
Indeed, participants felt reassured when they “connected” with health care services
for end-of-life care. Feeling “connected” with health care services centred on
engaging with health care services on their own terms:
I felt absolutely no control so I said to them [healthcare professionals] “I feel
a complete disconnect … [that] I’m kind of apprehensive about a lot of
things” … now I am connected and it’s a team effort. Now I’m able to sit
down and discuss it [end-of-life care] with them … make decisions when you
are in control [of care]. (Participant #31)
Overall, participants were reassured by healthcare professionals when they felt in
control of their care and when they secured services that would support them in
end-of-life care.
Resignation: Perceptions of life sustaining and of life ending
Participants’ need to be reassured about care arose primarily from feeling resigned
to MND. Participants struggled between living on with MND and resigning to death
but engaging with treatments that had the potential to prolong duration of distress
was unacceptable to most:
Participant: I suppose I just feel let me get on with it … [but] I mean there are
days when I don’t feel like living … hopefully there won’t be too much future
[left] …
Interviewer: Do you have any particular feelings about life-sustaining
treatment?
Participant: Dragging it out. Well I don’t see the point … I think people might
incorrectly believe that a life-sustaining treatment might lead to a general
improvement … the feeling of having something [like this] dragging on and
on. I don’t want that. (Participant #27, end-stage MND)
8
The pace of disease progression in MND alerted participants to their impending
death and some participants wanted to die before the final stage of MND because
they anticipated suffering at the end stage of MND:
Last night was the first time that I found it difficult to breathe … It panicked
me and I know that’s what's going to kill me. Em and that is going to be hard
to get through that … The diaphragm is going to slowly pack up. So I know it’s
not going to be nice … I would like to go [die] before that point. (Participant
#22)
Participants questioned the role of life-sustaining treatments (e.g., assisted
ventilation) and in some cases, even supportive care (e.g., nursing and allied
healthcare), in the context of the terminal nature of MND. Most participants
suggested that losing independence rendered life less meaningful and that lifesustaining interventions had the potential to prolong “suffering”. Of note,
participants perceived noninvasive ventilation and gastrostomy as life-sustaining
interventions:
I think I’d be dead [without gastrostomy feed and noninvasive ventilation] …
The RIG [gastrostomy feed] was put in before I needed it … I got this
[pointing to noninvasive ventilation] before I needed it ... everything was
anticipated ... [but] the whole thing [MND] is a nightmare, well the whole,
the whole concept [of living with MND] is a nightmare. (Participant #24, endstage ALS)
I felt I was rushed into it [gastrostomy feed]. I would have liked more time to
have thought about it … when you get to that stage, what’s the point … It’s
just a life line for the medics to put in food and drugs. … to sustain life
beyond what it should be. (Participant #10)
9
I do wonder a lot, I’m saying why are they [healthcare professionals] keeping
me alive for longer, why don’t they just let me fade away, you know the end
is going to be the same when it comes, so why prolong the suffering.
(Participant #32)
A small minority of participants (n=2) wished to live on indefinitely with high levels of
disability but no participant suggested in the course of interview that they would
seek invasive (long-term) ventilation to sustain life:
When you can’t breathe on your own, it’s game over. (Participant #21,
anarthric and quadriplegic)
Participants who spoke about euthanasia and/or physician-assisted suicide (n=6)
indicated that physician-assisted suicide was a reasonable care option for people
with MND, and suggested that most people with MND would at some point weigh up
the pros and cons of euthanasia and/or physician-assisted suicide:
I think you know, it gets to the stage where somebody’s life where you know,
there’s just nothing going on for them … pain all around … I would defy
anyone to say they wouldn't think of it. (Participant #11)
Participants associated palliative care with the dying phase only and few participants
related life-sustaining interventions in MND with a palliative care approach.
Participants associated palliative care with treatments that would alleviate suffering
at end-of-life but did not construe life-sustaining treatments as interventions to ease
potential “suffering”:
Interviewer: Some people choose life-sustaining treatment and others don’t.
Do you have any feelings or thoughts about that?
Participant: It would be staying alive under what conditions. Like you know, if
they were just keeping you alive … that Hawkins fella [man], sure he has it
10
since he was 22 and he is 75 now … But he’s all curled up in a chair. Ah I
would hate that …
Interviewer: So that wouldn't be acceptable to you?
Participant: No, even if I had his brain. (Participant #13)
Overall, participants were resigned to the terminal nature of MND and questioned
life-sustaining treatments for the sake of longevity alone..
Trust
Participants had a strong desire to place trust in healthcare professionals at all stages
of the disease. Participants varied in how much they trusted healthcare
professionals, and we found instances of mistrust:
I was going down to [private consultant neurophysiologist] in [regional
hospital] and he wasn’t saying anything to me, he was making a pile of
money … In fairness I thought this is [swear], nobody knows what’s wrong
with me and it’s going on and on … The whole thing was disjointed and the
worst of it, I was being kept in the dark … I’d lost confidence in the whole
[swear] thing [diagnosis]. (Participant #30)
We found many dimensions to trust in our data that shed light on how and why
participants trusted (or mistrusted) healthcare professionals. Participants placed
little if any trust in healthcare professionals who lacked empathy in the clinical
encounter but were trusting of healthcare professionals who were knowledgeable in
the field, who were personable and “human” in their approach and who reassured
them about their care:
It’s just the way she [consultant neurologist] kind of explained about the
motor neurone disease and everything and what’ll happen further down the
line … I said “how sure are you” and she said “we’re a hundred percent” so at
least she was straight up … I found her very easy to talk to … after a few
minutes you’d kind of open up … I’d be trusting her. (Participant #28)
11
Participants shared serious concerns about the viability of the Irish healthcare
system but remained reassured about their care when they encountered individual
healthcare professionals whom they trusted. Participant #7 explained:
Interviewer: Do you worry sometimes about later on, in terms of your care
services?
Participant: Not hugely, right, that goes back to the trusting element of my
nature … I’ve a very good friend and she happens to be in the hospice, she’s
actually in her final stages of cancer … I went to visit her … and it was a very
good experience from my point of view knowing down the line that I will be
using this service and it was good for me to experience people who are in a
caring role looking after the patients that were there.
Central to participants’ experience of ALS was loss including loss of control over their
lives. We found that participants trusted healthcare professionals who were
sensitive to participants’ loss and who understood how participants exerted control
in health care services to adapt to loss:
I was in a lot of trouble [difficulty mobilising] until she [community
occupational therapist] brought me the wheelchair even though I rejected it
at the start because I felt I didn't need that kind of thing … I hid it in the shed
outside for a while … I just hated the thought of being wheeled in that
(crying) but I've no bother now … It’s a process to get to that, that’s the
process you have to do yourself. I thought that end of it was very
professional the way it was left to me and it wasn’t forced on me … I trusted
her then. (Participant #8)
Overall, participants trusted healthcare professionals who reassured them and who
enabled them to be in control of their care.
Discussion
12
We identified that control, trust, and reassurance are key variables that shape how
people with MND engage with health care services as they resign themselves to the
terminal nature of MND. People with MND feel more reassured about their care
when they feel in control of care and they seek reassurances from healthcare
professionals about their care in the face of disease progression. People with MND
trust healthcare professionals who enable them to be in control of care and who
reassure them about their care. However, interventions that might prolong life
provoke anxiety for those who anticipate (or experience) “suffering”.
Our findings pertaining to reassurance and trust are new to the MND field. We knew
that existential issues were important to people with MND 20,21 and that people with
MND expected honesty and sensitivity from healthcare professionals 22 but we
discovered that reassurance and trust are key variables that shape how people MND
engage with health care services. Researchers have already reported on control in
MND. Cooney et al.23 found that that being in control of care is of central importance
to people with MND and that people with MND have a strong desire for self
determination in end-of-life care. We captured why people with MND feel they need
to be in control of care; namely to engage with services when they themselves
decide that services are needed to cope with loss.
Our findings resonate to some extent with those of other studies on service users’
preferences for / perceptions of end-of-life care24,25 and their expectations of
healthcare professionals in palliative care.26-29 Rodriguez and Young24 also found that
service users questioned the benefit of life-sustaining interventions in situations
where they perceived their quality of life as unacceptable. As in MND, people with
advanced cancer prefer to be in control over treatment decisions and end-of-life
care.25 People with other terminal illnesses (including cancer, heart failure, and
obstructive pulmonary disease) also seek to avoid unwanted life-sustaining support
and need to be reassured by healthcare professionals about end-of-life care.26 They
also have a need to trust healthcare professionals as they approach death26,27,
expect healthcare professionals to communicate openly with them26-29, value
honesty and sensitive sharing of information26-28 and have a strong desire to be
13
involved in the decisions about their care.27,29 Similar to our findings, Evans et al.27
found that service users’ trust (or mistrust) in healthcare professionals is calibrated
by how they perceive healthcare professionals’ approach towards them. Empathy
and genuine concern from healthcare professionals can engender trust in the service
user but a lack of sensitivity on behalf of service providers can diminish service users’
trust in service providers.27
Our findings shed light on MND service users’ perceptions of life-sustaining
treatments and of palliative care. We found that participants were resigned to death
because of the terminal nature of MND and they were averse to the notion of
prolonging life in MND for the sake of longevity alone. Participants nearing death
wanted to die soon. Participants did not associate palliative care with care before
the dying phase even though the care approach in MND is palliative.3,4 Few
participants associated noninvasive ventilation and gastrostomy feeding with
palliative care even though noninvasive ventilation and gastrostomy feeding
alleviate symptoms in MND.3 We also discovered that some participants construed
gastrostomy feeding as a life-sustaining intervention even though gastrostomy
feeding might not necessarily improve survival in MND.3
Our findings resonate with those of Cooney et al.23 on preferences for end-of-life
care in MND. Cooney at al. also found that people with MND wanted to avoid
interventions that had the potential to sustain life beyond the natural course of the
disease. Clinicians regularly advocate early initiation of noninvasive ventilation and
gastrostomy feeding to improve survival and/or to alleviate symptoms.3,30 However,
some participants in our study were averse to these interventions because they did
not perceive them as interventions to alleviate “suffering”.
We were attuned to how context shaped our substantive findings.15 We found that
categories of ‘trust’ and ‘reassurance’ were shaped in part by participants’
experiences of the national clinic. Participants’ high level of satisfaction with
healthcare professionals at a specialist MND clinic is consistent with that of MND
service users in specialist clinics in other countries.31-33 Participants’ experiences of
14
‘life-sustaining’ interventions are also contextualised within the Irish healthcare
system. People with MND in Ireland use noninvasive ventilation but invasive
ventilation is not readily available to them. People with MND in countries that
routinely offer invasive ventilation (e.g., Japan, USA) have reported conflicting
emotions about invasive ventilation.10,34
Strengths and Limitations
The findings arise from qualitative interviews with people with MND in one country
and so our findings are not representative of the global MND population. Each
participant was interviewed on one occasion only and so we did not capture
participants’ experiences of healthcare services over time. However, at a substantive
level, we have identified key variables that shape how people with MND engage with
health care services. The substantive findings also enable comparison with other
contexts.15
Conclusion
Our findings have important implications for practice. People with MND want to
engage with healthcare professionals on their own terms and value meaningful
relationships with healthcare professionals. We were attuned to both micro and
macro contexts15 of participants’ experiences of services and found that participants’
concerns were alleviated when they trusted healthcare professionals and felt
reassured by them. We therefore postulate that trust and reassurance for MND
service users operate predominantly at the micro-level (i.e. clinical encounter) of
healthcare experiences.
Practice parameters in ALS care3,30,35 are based on the consensus of experts in the
field and on evidence which for the most part, does not include the service user
perspective on health care services. Service users’ perceptions and understandings
of ‘palliative’ and of ‘life-sustaining’ interventions in MND might differ from those of
healthcare professionals who render services to them. People with MND do not
necessarily equate alleviation of symptoms with alleviation of suffering. Research
into how people with MND construe “suffering” is required.
15
Acknowledgements
We are sincerely thankful to all participants for sharing their experiences with us. We
also thank the Health Research Board (HRB) of Ireland for funding this study.
Author contributions
GF and VT designed the study with OH as advisor. GF sampled participants,
conducted the interviews, and analysed the data. VT guided sampling procedures
and interrogated the analysis. OH facilitated acquisition of the data. GF wrote the
article and VT and OH commented on first and subsequent drafts. All authors
approved the final draft.
Funding
This study was funded by the Health Research Board (HRB) of Ireland [HPF/2011/1].
Conflict of Interest
The authors declare that there is no conflict of interest.
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